Accepted for/Published in: Journal of Medical Internet Research
Date Submitted: Aug 31, 2021
Date Accepted: May 4, 2022
A Remote Digital Monitoring Platform to Assess Cognitive and Motor Symptoms in Huntington’s Disease: Cross-Sectional Validation Study
ABSTRACT
Background:
Remote monitoring of Huntington’s disease (HD) signs and symptoms using digital technologies may enhance early clinical diagnosis and tracking of disease progression, guide treatment decisions and monitor response to disease-modifying agents. Several recent studies in neurodegenerative diseases have demonstrated the feasibility of digital symptom monitoring.
Objective:
To evaluate a novel smartwatch- and smartphone-based digital monitoring platform to remotely monitor signs and symptoms of HD.
Methods:
This analysis aimed to determine cross-sectional validity of the Roche HD Digital Monitoring Platform over a 2-week interval. Key criteria assessed were feasibility, evaluated by adherence and quality control (QC) failure rates, test-retest reliability, known-groups validity, and convergent validity of sensor-based measures with existing clinical measures. Data from three studies were used: the pre-drug screening phase of an open-label extension study evaluating tominersen (NCT03342053) and two untreated cohorts—HD Natural History Study (NCT03664804) and Digital-HD. Across these studies, controls and individuals with premanifest or manifest HD completed six motor and two cognitive tests at home and in clinic.
Results:
Adherence to active tests was good-to-excellent. Most sensor-based features showed good-to-excellent test-retest reliability (intraclass correlation coefficient, 0.65–0.98) and low QC failure rates. Good overall convergent validity of sensor-derived features to Unified HD Rating Scale outcomes and good overall known-groups validity between controls, premanifest and manifest participants were observed.
Conclusions:
These results show the potential of the HD Digital Monitoring Platform to provide reliable, valid, continuous remote monitoring of HD symptoms, facilitating the evaluation of novel treatments and enhanced clinical monitoring and care for individuals with HD. Clinical Trial: Open-label extension study evaluating tominersen ClinicalTrials.gov number, NCT03342053; HD Natural History Study ClinicalTrials.gov number, NCT03664804
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